REVIEW ARTICLE

Juvenile Osteochondrosis of the Cuneiform Bones

N. K. Sferopoulos

Department of Pediatric Orthopaedics, “G. Gennimatas” Hospital, Thessaloniki, Greece

ABSTRACT

Osteochondrosis of the tarsal and metatarsal bones is frequent in children. However, involvement of a cuneiform bone is a
very rare entity with only scant reports in the world medical literature. The purpose of this paper is to retrospectively estimate
the incidence rate of juvenile osteochondrosis of the cuneiform bones at our institution and to review the relevant publications.

Key words: Cuneiform, juvenile, osteochondrosis

REVIEW Osteochondrosis is defined as an idiopathic syndrome

characterized by abnormal endochondral ossification occurring

P
edal osteochondrosis may cause pain and/or limping
in children. The most commonly affected sites
include the calcaneal apophysis (Sever’s disease), the
tarsal navicular (Köhler’s disease), the second metatarsal
head (Freiberg’s infraction or Köhler’s disease II), and the
apophysis of the fifth metatarsal base (Iselin’s disease).
Osteochondrosis of the cuneiform bones is apparently a very
rare entity.[1-5]
In 1930, Harbin and Zollinger were the first to report a
patient with osteochondrosis of the medial (first or internal)
cuneiform.[6] Buchman, in 1933, described two children with
osteochondrosis involving the medial cuneiform.[7] Haboush,
Meilstrup, and O’Donoghue et al. reported, respectively,
a case with bilateral involvement of the medial cuneiform
and abnormal navicular on plain radiographs.[8-10] Hicks and
Smyth were the first to report a unilateral osteochondrosis of
the intermediate cuneiform.[11,12] Breck was able to diagnose
11 patients with osteochondrosis of the medial cuneiform and
9 with osteochondrosis of the intermediate cuneiform among
1000 patients with osteochondroses.[13] Since then, nine more
children with osteochondrosis of the medial and six with
osteochondrosis of the intermediate cuneiform bones were
reported in the English literature.[14-29] Mubarak was the only
one to document a case with osteochondrosis of the lateral
cuneiform.[30]
in a previously normal growth mechanism. It is usually due
to a vascular insult, leading to secondary osteonecrosis. The
precise etiology of osteochondrosis remains unknown since
multiple contributing etiologic factors, such as microtrauma,
rapid growth, genetic, and/or hormonal factors, may be
involved. It may be prudent to consider that the early onset
of ossification of the cuneiform bones, before the mechanical
stresses of walking on the developing center of ossification
are incurred, may be related to the low incidence of cuneiform
osteochondrosis in children. The radiographic changes
closely resemble those of osteonecrosis (avascular, aseptic, or
ischemic bone necrosis). The term osteochondrosis is preferred
than that of osteochondritis, which more specifically refers to
infection or inflammation of bone and cartilage. Although
the term osteochondrosis is the most precise nomenclature to
describe the lesion, other definitions, such as osteochondritis,
osteonecrosis, and avascular necrosis, are also occasionally
used.[25,26,31-35]
The average age of the patients in the previously published
reports ranged from 2.5 to 8 years, although it mainly
appears between 4 and 6 years of age, boys were more
frequently affected and cases with a bilateral involvement
were more commonly reported. Pain and/or limping were
the predominant symptoms in the majority of cases, while
in the asymptomatic cases, the diagnosis was based on the

Address for correspondence:

N. K. Sferopoulos, P. Papageorgiou 3, 546 35, Thessaloniki, Greece. Phone: 00302310963270.
E-mail: 
https://doi.org/10.33309/2638-7654.020201 www.asclepiusopen.com
© 2019 The Author(s). This open access article is distributed under a Creative Commons Attribution (CC-BY) 4.0 license.

Clinical Research in Pediatrics  •  Vol 2  •  Issue 2  •  2019 1

 Sferopoulos: Cuneiform osteochondrosis

coincidental radiographic examination for foot injuries or
abnormalities. The most common finding of the physical
examination is tenderness over the related cuneiform bone.
However, most commonly the clinical symptoms and the
physical findings are nonspecific. Laboratory studies are
usually normal. The radiographic changes usually are
unremarkable at the beginning, although sites of decreased
bone density of the affected cuneiform may occasionally
be diagnosed. A decrease in size and marginal irregularity
as well as sclerotic areas and structural collapse due to
osteonecrosis may be evident on plain films, during the
progress of the disease. Magnetic resonance imaging may be
useful on the rare occasions that differential diagnosis from
symptomatic bipartite medial cuneiform, osteomyelitis, or
bone tumors is indicated. Osteochondrosis of the cuneiform
bones is occasionally defined as a variant of normality,
considering its benign self-limited nature. The disorder
necessitates only symptomatic treatment. Medical therapy
for pain control and physical activity restriction is the only
required treatment modality. The clinical findings as well as
the radiographic abnormalities resolve spontaneously, in all
patients, irrespective of weight-bearing and immobilization
treatment. Aggressive surgical treatment is unnecessary
and potentially harmful to the patient. A short leg cast for
6-8 weeks accelerates the resolution of symptoms, although
long-term outcomes are favorable regardless of treatment.
A biopsy is not recommended for the diagnosis unless
there is the need to rule out infection, especially the most
common subacute form of osteomyelitis, Brodie’s abscess,
or malignancy.[25-30,36-39]
a spiral fracture of the right tibia with minimal displacement.
Although there was no specific clinical evidence of a foot injury,
dorsal-plantar and oblique foot radiographs were performed.
A translucent line was evident on the distal pole of the medial
cuneiform which had an irregular outline and cystic-like sites
of decreased bone density. Sclerotic areas associated with
sites of decreased bone density of the intermediate cuneiform
were also diagnosed. There was also decreased bone density
of the medial aspect of the lateral cuneiform. The radiographic
findings were considered to be indicative of osteochondrosis
of all cuneiform bones [Figure 1], despite no clinical findings
detected from the physical examination or reported in the
patient’s history from his parents.
Although pedal osteochondrosis occurring at multiple sites
has been previously reported,[8,10,14,16,28,45-48] concomitant
ipsilateral osteochondrosis of all the cuneiform bones has not
been previously described.
The second patient was a 5-year-old boy who was referred
after an in-home injury of his right foot. There was no evidence
of a fracture line on the dorsal-plantar foot radiograph.
The radiographic findings of marginal irregularity and the
sclerotic areas associated with sites of decreased bone density
of the medial cuneiform were considered to be indicative of
osteochondrosis of the medial cuneiform [Figure 2].
Finally, the third patient was a 6-year-old boy who was referred
after a school injury of his left foot. There was no evidence of a

The differential diagnosis may be confused in post-traumatic

cases, following acute or stress fractures, or in patients
with a bipartite cuneiform, which are all extremely rare in
childhood.[37,40,41]

Osteonecrosis of the cuneiform is an unusual pathology to

the foot in adults. Risk factors in the case of non-traumatic
osteonecrosis include the use of corticosteroids, smoking,
alcohol, sickle cell anemia, radiation treatments, as well as
rheumatologic, hematologic, and metabolic disorders.[42-44]

The radiographs of children, younger than 14 years of age,

with foot injuries or disorders that were referred to both the
emergency and the outpatient department between January
2000 and December 2015, were retrospectively evaluated by
the author from the hospital radiographic database. From a
total amount of approximately 1500 children, no cases with a
diagnosis of osteochondrosis of the cuneiform were identified.
From the retrospective revue of the radiographs, three cases
with radiographic findings consistent with osteochondrosis
of the cuneiform bones were identified. Figure 1: A  4-year-old boy with radiographic findings,
consistent with osteochondrosis, involving the full extent of
The first patient was a 4-year-old boy who injured his right leg the medial and intermediate cuneiform bones and the medial
in a road traffic accident. Radiographic examination indicated aspect of the lateral cuneiform bone

2 Clinical Research in Pediatrics  •  Vol 2  •  Issue 2  •  2019

 Sferopoulos: Cuneiform osteochondrosis
Figure 2: A  5-year-old boy with radiographic findings,
consistent with osteochondrosis, involving the medial
cuneiform bone
Figure 3: A  6-year-old boy with radiographic findings,
consistent with osteochondrosis, involving the medial
cuneiform bone
fracture line on the dorsal-plantar and oblique foot radiographs.
The radiographic findings of marginal irregularity and the
sclerotic areas associated with sites of decreased bone density
of the medial cuneiform were considered to be indicative of
osteochondrosis of the medial cuneiform [Figure 3].
It is apparent that in all three patients, the diagnosis of
osteochondrosis of the cuneiform bones was retrospectively
Clinical Research in Pediatrics  •  Vol 2  •  Issue 2  •  2019
made from the evaluation of the radiographic examination
following an ipsilateral leg or foot injury. It may be reasonable
to consider that the patients were asymptomatic or minimally
symptomatic before the traumatic insults. It may be prudent to
realize that the incidence of osteochondrosis of the cuneiform
may be higher than the reported one in the world literature
since a great percentage of patients remain undiagnosed due
to the mild symptomatic course of the disorder as well as to
the age distribution, provided that the disorder usually affects
pre-school and early school-age children.
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How to cite this article: Sferopoulos NK. Juvenile
Osteochondrosis of the Cuneiform Bones. Clin Res
Pediatr 2019;2(2):1-4.